The DOI matches its Crossref record; 124 of the 125 references it cites were found in a registry or an index.
Tubby-like protein 3 (TULP3) regulates patterning in the mouse embryo through inhibition of Hedgehog signaling
Open accessAbstract
Tubby-like protein 3 (TULP3) is required for proper embryonic development in mice. Disruption of mouse Tulp3 results in morphological defects in the embryonic craniofacial regions, the spinal neural tube and the limbs. Here, we show that TULP3 functions as a novel negative regulator of Sonic hedgehog (Shh) signaling in the mouse. In Tulp3 mutants, ventral cell types in the lumbar neural tube, which acquire their identities in response to Shh signaling, are ectopically specified at the expense of dorsal cell types. Genetic epistasis experiments show that this ventralized phenotype occurs independently of Shh and the transmembrane protein Smoothened, but it is dependent on the transcription factor Gli2. The ventralized phenotype is also dependent on the kinesin II subunit Kif3A, which is required for intraflagellar transport and ciliogenesis. In addition, TULP3 is required for proper Shh-dependent limb patterning and for maintaining the correct balance between differentiation and proliferation in the neural tube. Finally, the localization of TULP3 to the tips of primary cilia raises the possibility that it regulates the Hedgehog pathway within this structure.
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“Interestingly, mutations in the genes encoding TULP3 or IFT-A subunits phenocopy Gpr161 mutants by increasing HH signalling in the caudal neural tube 58, 128–131.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [130]
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Cites
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Vertebrate Smoothened functions at the primary cilium
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Patched1 Regulates Hedgehog Signaling at the Primary Cilium
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Hedgehog signalling in the mouse requires intraflagellar transport proteins
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Items 101–102 of 102
Repušić, D., Korolija, M., Livun, A., Svaguša, T., Belužić, R., Tomaić, V., Karlić, R., & Vlahoviček, K. (2026) · Journal of Medical Virology · doi:10.1002/jmv.70984
Primary cilium-mediated mechanisms of cyst formation in polycystic kidney disease
Qian, F., & Mukhopadhyay, S. (2026) · Nature Reviews Nephrology · doi:10.1038/s41581-026-01139-0
Metadata
- Authors
- Norman, R. X., Ko, H. W., Huang, V., Eun, C. M., Abler, L. L., Zhang, Z., Sun, X., & Eggenschwiler, J. T.
- Affiliations
- Princeton University (US)
- University of Wisconsin–Madison (US)
- Corresponding author
- Eggenschwiler, Jonathan T.
- Venue
- Human Molecular Genetics
- Publisher
- Oxford University Press (OUP)
- Published
- 2009-03-12
- Volume
- 18
- Issue
- 10
- Pages
- 1740-1754
- ISSN
- 0964-6906 (ISSN-L)
- 1460-2083
- PMC
- PMC2671991
- OpenAlex
- W2170193319
- Cited by
- 103
- References
- 125
- Citations by year
- 2026: 3 · 2025: 6 · 2024: 9 · 2023: 5 · 2022: 6 · 2021: 7 · 2020: 10 · 2019: 6 · 2018: 5 · 2017: 5 · 2016: 7 · 2015: 5 · 2014: 4 · 2013: 4 · 2012: 9
- FWCI
- 2.82
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Human Molecular Genetics (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Funders
- New Jersey Commission on Spinal Cord Research
- National Institutes of Health (IA 52242)
- Directorate for Biological Sciences
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (R01HD045522, HD045522, HD050761, R01HD050761)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Hedgehog Signaling Pathway Studies
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Keywords
- TULP3, Sonic hedgehog signaling, Hedgehog signaling, neural tube patterning, GLI2, KIF3A, kinesin-2, intraflagellar transport, ciliogenesis, primary cilia, limb patterning, craniofacial defects, Smoothened, mouse embryo
- MeSH terms
- Animals, Embryo, Mammalian, Female, Humans, Male, Mice, Inbred C3H, Mice, Transgenic, Mutation, Proteins, Spinal Cord, Signal Transduction, Down-Regulation, Gene Expression Regulation, Developmental, Body Patterning, Intercellular Signaling Peptides and Proteins, Embryonic Development, Intracellular Signaling Peptides and Proteins, Mice, Hedgehog Proteins, Neural Tube
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